Silberstein L E, Abrahm J, Shattil S J
Transfusion. 1987 May-Jun;27(3):234-7. doi: 10.1046/j.1537-2995.1987.27387235627.x.
This study describes the response to therapeutic plasma exchange in a 60-year-old man with Waldenström's macroglobulinemia who developed a clinically severe bleeding disorder with laboratory features characteristic of acquired von Willebrand's disease. The patient's plasma levels of factor VIII coagulant activity, von Willebrand's factor antigen, and ristocetin cofactor activity were all less than 15 percent of normal, and the bleeding time was more than 20 minutes. In vitro studies did not demonstrate an inhibitor to factor VIII/von Willebrand factor, nor was a precipitating antibody to von Willebrand's factor antigen found in the patient's plasma. Neither infusions of cryoprecipitate nor combination chemotherapy corrected the clinical or laboratory abnormalities. In contrast, plasma exchange corrected the in vitro coagulation abnormalities and was effective in preventing surgical hemorrhage and controlling severe mucosal bleeding on 11 separate occasions. The current case demonstrates that the clinical and laboratory abnormalities in a patient with acquired von Willebrand's disease can be corrected completely by plasma exchange; it is recommended therefore that plasma exchange be considered as a mode of therapy in symptomatic patients with this disorder.
本研究描述了一名60岁患有华氏巨球蛋白血症的男性患者对治疗性血浆置换的反应,该患者出现了临床上严重的出血性疾病,其实验室特征符合获得性血管性血友病。患者血浆中凝血因子VIII促凝活性、血管性血友病因子抗原和瑞斯托霉素辅因子活性均低于正常水平的15%,出血时间超过20分钟。体外研究未显示存在针对凝血因子VIII/血管性血友病因子的抑制剂,患者血浆中也未发现针对血管性血友病因子抗原的沉淀抗体。输注冷沉淀和联合化疗均未能纠正临床或实验室异常。相比之下,血浆置换纠正了体外凝血异常,并在11次不同情况下有效预防了手术出血和控制了严重的黏膜出血。目前的病例表明,获得性血管性血友病患者的临床和实验室异常可通过血浆置换完全纠正;因此,建议将血浆置换作为该疾病有症状患者的一种治疗方式。